Infant Salmonella enterica Meningitis: A Rare Case Report and Review of Literature.

Meningitis caused by Salmonella enterica can be a fatal condition that is more common in low- and middle-income countries and uncommon in infants. This case of a 2-month-old male infant reported Salmonella meningitis symptoms, such as fever, irritability, altered sensorium, and diarrhoea. Clinical examination revealed bulging anterior fontanelles, dehydration, and sunken eyes. Screening for normal hearing, cranial ultrasound, and magnetic resonance imaging (MRI) revealed no brain abnormalities. A cerebrospinal fluid (CSF) culture revealed gram-negative Salmonella enterica bacilli. Treatment with meropenem and ampicillin was initiated after antibiotic susceptibility testing showed sensitivity. The patient's cerebrospinal fluid parameters and bacterial growth improved after antibiotic therapy. Two weeks later, the baby was neurologically healthy and discharged. Paediatricians should be aware that Salmonella enterica can cause meningitis in children with non-specific symptoms.

Economic impact of metagenomic next-generation sequencing versus traditional bacterial culture for postoperative central nervous system infections using a decision analysis mode: study protocol for a randomized controlled trial.

IMPORTANCE: Diagnosing and treating postoperative central nervous system infections (PCNSIs) remains challenging due to the low detection rate and time-consuming nature of traditional methods for identifying microorganisms in cerebrospinal fluid. Metagenomic next-generation sequencing (mNGS) technology provides a rapid and comprehensive understanding of microbial composition in PCNSIs by swiftly sequencing and analyzing the microbial genome. The current study aimed to assess the economic impact of using mNGS versus traditional bacterial culture-directed PCNSIs diagnosis and therapy in post-neurosurgical patients from Beijing Tiantan Hospital. mNGS is a relatively expensive test item, and whether it has the corresponding health-economic significance in the clinical application of diagnosing intracranial infection has not been studied clearly. Therefore, the investigators hope to explore the clinical application value of mNGS detection in PCNSIs after neurosurgery.

[A case of tuberculous meningitis diagnosed early by direct Loop-Mediated Isothermal Amplification (LAMP) method using centrifuged medium of cerebrospinal fluid culture].

Tuberculous meningitis (TBM) is a central nervous system infection with a high mortality rate and requires early diagnosis and treatment. Identification of Mycobacterium tuberculosis in the cerebrospinal fluid is of primary importance in the diagnosis of TBM, however, conventional methods have some disadvantages: Rapid results tests such as smear and regular PCR method do not have sufficient diagnostic sensitivity; Nested PCR, which is one of the most sensitive tests, is not available in all facilities; Culture tests require a long period of 4-8 weeks for results. Here we report a case of TBM, diagnosed 14 days earlier than culture test by direct Loop-Mediated Isothermal Amplification (LAMP) method using centrifuged medium of cerebrospinal fluid (day 18) culture. The method we used here is simple, widely available, and considered to be useful for early detection of TBM.

Clinical Characteristics and Risk Factors for Mortality in Cryptococcal Meningitis: Evidence From a Cohort Study.

Introduction: Despite advances in the diagnosis and management, cryptococcal meningitis (CM) is still associated with high mortality due to insufficient knowledge about clinical characteristics and risk factors for poor outcomes. The aim of the present study is to provide additional evidence for regarding clinical characteristics, diagnosis, and factors associated with increased risk of mortality in CM patients. Methods: In this cohort study, we included eligible patients consecutively admitted to West China Hospital between January 2009 and December 2018. The clinical characteristics and diagnosis method of cerebrospinal fluid culture and India ink stain were analyzed. Independent risk factors were identified by a multivariable logistic regression. Results: A total of 186 CM patients were included in the analysis. After a 1-year follow-up, 63 patients had died. Headache is the most common presenting symptom (97.3%), followed by vomiting (72%), fever (71.5%), altered consciousness (45.7%), abnormal vision (32.8%), and seizure (15.1%). Older age, altered consciousness or seizures, lower white blood cell count or total protein in cerebrospinal fluid (CSF), and unidentified CSF cryptococcal antigen (CrAg) are all factors associated with increasing risk of death (P < 0.05). We also found a dose-dependent trend between the number of symptoms and risk of death (trend p < 0.001). Multivariate logistic regression revealed that age (P = 0.004, OR = 1.042, 95% CI 1.013-1.071), seizure (P = 0.025, OR = 3.105, 95% CI 1.152-8.369), altered consciousness (P < 0.001, OR=6.858, 95% CI 3.063-15.38), and unidentified CSF CrAg are the independent prognostic factors. In addition, we observed that diagnosis of 28.5% and 22.5% CM could not be established by a single testing of CSF India ink stain or culture, respectively. Use of multiple testing methods or combination of the two assays increases the detection rate. Conclusion: Our data show that older age, seizures, altered consciousness, and an inability to detect CSF CrAg are the independent risk factors of death within 1 year in CM patients. Moreover, we recommend use of multiple testing methods with CSF culture and India ink stain. Combined testing with both assays should be considered for initial CM diagnosis.

Intramedullary abscess at thoracolumbar region transmitted from infected dermal sinus and dermoid through retained medullary cord.

BACKGROUND: A retained medullary cord (RMC) is a relatively newly defined entity of closed spinal dysraphism that is thought to originate from regression failure of the medullary cord during secondary neurulation. A congenital dermal sinus (CDS) may provide a pathway for intraspinal infections such as repeated meningitis. Intramedullary abscesses are the rarest but most serious complication of a CDS. CASE DESCRIPTION: We treated a female infant with an intramedullary abscess in the thoracolumbar region, which was caused by infection of the CDS. Surgery revealed that the cord-like structure (C-LS) started from the cord with the intramedullary abscess, extended to the dural cul-de-sac, and further continued to the CDS tract and skin dimple. The boundary between the functional cord and the non-functional CL-S was electrophysiologically identified, and the entire length of the C-LS (the RMC) with an infected dermoid cyst was resected. As a result, the abscess cavity was opened and thorough irrigation and drainage of the pus could be performed. Histopathological examination of the C-LS revealed an infected dermoid cyst and abscess cavity with keratin debris in the fibrocollagenous tissue. The abscess cavity had a central canal-like ependymal lined lumen (CCLELL), with surrounding glial fibrillary acidic protein (GFAP)-immunopositive neuroglial tissues. CONCLUSION: We demonstrated that the transmission of an infection through the RMC was involved in the development of the intramedullary abscess. A good postoperative outcome was obtained because a terminal ventriculostomy for pus drainage could be achieved by excising the nonfunctional RMC.

Cerebrospinal fluid culture-positive bacterial meningitis increases the risk for neurologic damage among neonates.

OBJECTIVE: This study aimed to compare the clinical features and outcomes of neonatal bacterial meningitis (NBM) between patients with positive and negative cerebrospinal fluid (CSF) cultures and determine the risk factors for CSF culture-positive NBM. METHODS: We retrospectively reviewed the medical records of all patients with NBM. Perinatal clinical data, laboratory results, and cranial radiographs were obtained. RESULTS: Among the 186 neonates who met the inclusion criteria. The risk factors for positive CSF culture results were analysed using multiple logistic regression. The multivariable logistic regression analysis showed that the possible risk factors of NBM with positive CSF culture in this study were: Length of fever [OR = 1.126; 95% CI (0.999-1.268)], Neurologic symptoms [OR = 3.043; 95% CI (1.164-7.959)], Cerebrospinal fluid protein [OR = 1.001; 95% CI (1.000-1.001)]. Cases of NBM with a longer duration of fever, more neurologic symptoms, and higher levels of CSF protein were more likely to demonstrate positive results on CSF culture. CONCLUSION: Cases of NBM with CSF culture-positive results were more likely to have severe clinical manifestations and develop more serious neurologic damage. Patients with NBM who have longer durations of fever, more neurologic symptoms, and higher levels of CSF protein were more likely to have CSF culture-positive results, who should be followed up more closely.Key MessageBacterial meningitis is clinically defined as a serious inflammation of meningitis, usually caused by a variety of bacterial infections that may leave sequelae and long-term complications and high mortality rates. Early diagnosis is often difficult, particularly when the patient has been treated with antimicrobials.

Negative Cultures for Cerebrospinal Fluid Samples taken from the Myelomeningocele Sac on the Day of Repair.

OBJECTIVE: Our goal was to evaluate the intraoperative laboratory analysis of cerebrospinal fluid (CSF) aspirated from the myelomeningocele (MMC) sac prior to the repair to determine if there was bacterial growth in the culture. METHODS: This was a retrospective analysis of the CSF cultures of 45 MMC patients operated on during the years of 2002 to 2013 at the University Pediatric Hospital. Before repairing the defect, the sac area was cleaned and three milliliters of CSF were drawn and sent for analysis for red blood cells, white blood cells, glucose level, protein level, chloride level, gram stain and culture. The CSF sample results were analyzed for irregularities in the values before proceeding with placement of a ventricular shunt. RESULTS: All the CSF samples that were studied had at least 1 abnormal value in their results, even though none grew any pathogens in the cultures analyzed. CONCLUSION: Upon CSF analysis, we found increased levels of CSF protein and other abnormal values in this population; however, none of the cultures grew any pathogens. This finding is an important tool in the evaluation of the possible etiologies of and therapeutic approaches for future shunt problems in this group of patients.

A systematic review of cases of meningitis in the absence of cerebrospinal fluid pleocytosis on lumbar puncture.

BACKGROUND: Definitive diagnosis of meningitis is made by analysis of cerebrospinal fluid (CSF) culture or polymerase chain reaction (PCR) obtained from a lumbar puncture (LP), which may take days. A timelier diagnostic clue of meningitis is pleocytosis on CSF analysis. However, meningitis may occur in the absence of pleocytosis on CSF. Areas of Uncertainty: A diagnosis of meningitis seems less likely without pleocytosis on CSF, leading clinicians to prematurely exclude this. Further, there is little available literature on the subject. METHODS: Ovid/Medline and Google Scholar search was conducted for cases of CSF culture-confirmed meningitis with lack of pleocytosis. Inclusion criterion was reported cases of CSF culture-positive or PCR positive meningitis in the absence of pleocytosis on LP. Exclusion criteria were pleocytosis on CSF, cases in which CSF cultures/PCR were not performed, and articles that did not include CSF laboratory values. RESULTS: A total of 124 cases from 51 articles were included. Causative organisms were primarily bacterial (99 cases). Outcome was reported in 86 cases, 27 of which died and 59 survived. Mortality in viral, fungal and bacterial organisms was 0, 56 and 31%, respectively. The overall percentage of positive initial CSF PCR/culture for viral, fungal and bacterial organisms was 100, 89 and 82%, respectively. Blood cultures were performed in 79 of the 124 cases, 56 (71%) of which ultimately cultured the causative organism. In addition to bacteremia, concomitant sources of infection occurred in 17 cases. CONCLUSIONS: Meningitis in the absence of pleocytosis on CSF is rare. If this occurs, causative organism is likely bacterial. We recommend ordering blood cultures as an adjunct, and, if clinically relevant, concomitant sources of infection should be sought. If meningitis is suspected, empiric antibiotics/antifungals should be administered regardless of initial WBC count on lumbar puncture.

Development of a TaqMan Array card to target 21 purulent meningitis-related pathogens.

BACKGROUND: Purulent meningitis (PM) is a serious life-threatening infection of the central nervous system (CNS) by bacteria or fungi and associated with high mortality and high incidence of CNS sequelae in children. However, the conventional cerebrospinal fluid (CSF) culture method is time-consuming and has a low sensitivity. METHODS: Our study developed a real-time PCR-based purulent meningitis-TaqMan array card (PM-TAC) that targeted 21 PM-related pathogens and could produce results within 3 h. Primers and probes were adapted from published sources possibly. The performance of them were evaluated and optimized and then they were spotted on TAC. RESULTS: The PM-TAC showed a sensitivity and specificity of 95 and 96%, respectively. For all of the 21 targeted pathogens, the PM-TAC assay had a LOD ranging from 5 copies/reaction to 100 copies/reaction, an intra-assay variation of 0.07-4.45%, and an inter-assay variation of 0.11-6.81%. Of the 15 CSF samples collected from patients with PM after empiric antibiotic therapies, the positive rate was 53.3% (8/15) for our PM-TAC assay but was only 13.3% (2/15) for the CSF culture method. Of the 17 CSF samples showing negative CSF culture, the PM-TAC assay identified a case of Neisseria meningitidis infection. Furthermore, all of the 10 CSF samples from patients without CNS infection showed negative for the PM-TAC assay. CONCLUSIONS: Our PM-TAC assay also demonstrated that the pathogen loads in the CSF samples correlated with the severity of PM. Thus, the PM-TAC may be helpful to improve the prognosis of PM and clinical outcomes from antibiotic therapies.

Changing Pattern of Childhood Epidemic Cerebrospinal Meningitis in North-Western Nigeria.

INTRODUCTION: Nigeria lies within the meningitis belt which extends from the Gambia, Senegal through Nigeria to Eritrea; however, outbreaks have been shown to extend further south involving countries such as Angola and Namibia. Epidemic outbreaks are often recorded every 8-12 years averaging in a 10 yearly circle however endemic cases still occurs. MATERIALS AND METHODS: The study was retrospective; all results of cerebrospinal fluid (CSF) samples of children with cases of meningitis from January 2010 to December 2010 were collected from the register of the microbiology laboratory of General Hospital Gusau. Relevant information such as their age, sex, CSF macroscopy/microscopy reports, latex particle agglutination test report, and CSF culture report were retrieved and entered into a pro forma. RESULTS: There were 89 (73%) males and 33 (27%) females with male to female ratio of 2.7:1. The age ranged from 2 months to 14 years; the mean was 6.27 ± 4.00 years. Meningitis was mostly recorded from January to April. W135 was the most common serotype identified. Majority of the samples (54) which were nonreactive for any of the tested antigens had clear CSF (36), while among those that reacted; the W135 group had a high proportion of cases that had turbid CSF (44); (Fisher's exact test = 30.650, P = 0.000). Majority of the samples (99) had no cell count; although those of the W135 group had higher cell counts followed by those in the nonreactive group (Fisher's exact test = 11.226, P = 0.181). CONCLUSION: Meningitis was highest between January and April, and W135 was the most common serotype.

Características clínicas, epidemiológicas y manejo terapéutico de la meningitis pediátrica en dos instituciones de Medellín, Colombia / Clinical, epidemiological characteristics and therapeutic management of pediatric meningitis in two institutions of Medellin, Colombia

Resumen Introducción: La meningitis es una emergencia médica que requiere diagnóstico y tratamiento oportuno para evitar complicaciones. Objetivo: Determinar las características clínicas, epidemiológicas y el manejo terapéutico de meningitis en la primera infancia. Materiales y métodos: Estudio transversal que evaluó menores hasta los seis años de edad con meningitis bacteriana o aséptica en dos centros de Medellín, 2010 - 2013. Se recolectaron características clínicas, paraclínicas y terapéuticas. Las variables cualitativas se describieron mediante frecuencias absolutas y relativas y las cuantitativas con mediana y rango intercuartílico. Se exploró diferencias en las características de los pacientes según grupo de edad, mediante la prueba Chi-cuadrado o Fisher. Resultados: De 56 pacientes, 33 (58,9%) eran del sexo masculino; 26 (46,4%) presentaron meningitis bacteriana, 20 (35,7%) aséptica y 10 (17,9%) indeterminada; 36 (64,3%) fueron mayores de dos meses, con manifestaciones clínicas inespecíficas (fiebre e irritabilidad). Se aislaron principalmente microorganismos Gram negativos en hemocultivos y Gram positivos en líquido cefalorraquídeo. El tratamiento antibiótico más frecuente fue cefalosporinas de tercera-generación en 13 (65%) menores de 2 meses y 28 (77,8%) mayores. Fallecieron dos pacientes y seis presentaron complicaciones. Conclusiones: la meningitis bacteriana fue la más frecuente en menores de un año. El tratamiento combinado es ideal para garantizar un adecuado cubrimiento y evitar complicaciones.

Abstract Introduction: Meningitis is a medical emergency that requires early diagnosis and treatment to avoid complications. Objective: To determine the clinical, epidemiological characteristics and therapeutic management of meningitis in early childhood. Materials and methods: A cross-sectional study, which evaluated children up to six years old with bacterial or aseptic meningitis from 2010 to 2013, was conducted in two centers in Medellin. Clinical, paraclinical and therapeutic characteristics were collected. The qualitative variables were described by absolute and relative frequencies, and the quantitative ones with median and interquartile range. Differences in patient characteristics according to age group were explored, using the Chi-square or Fisher test. Results: From the 56 patients studied, 33 (58.9%) were male; 26 (46.4%) showed bacterial meningitis, 20 (35.7%) aseptic one and 10 (17.9%) indeterminate one; 36 (64.3%) were older than two months, with non-specific clinical manifestations (fever and irritability). Gram-negative microorganisms were mainly isolated in blood cultures and Gram positive in cerebrospinal fluid. The most common antibiotic treatment was third-generation cephalosporins in 13 (65%) children who were less than 2 months and 28 (77.8%) in older ones. Two patients died and six children presented complications. Conclusions: Bacterial meningitis was the most frequent in children under one year old. Combined treatment is ideal to ensure adequate coverage and avoid complications.

[Aetiology and outcomes of potentially serious infections in febrile infants less than 3 months old]. / Etiología y evolución de las infecciones potencialmente graves en lactantes menores de 3 meses febriles.

BACKGROUND: Recent studies have shown changes in the aetiology of serious bacterial infections in febrile infants ≤ 90 days of age. The aim of this study was to describe the current microbiology and outcomes of these infections in Spain. MATERIAL AND METHODS: Sub-analysis of a prospective multicentre study focusing on febrile infants of less than 91 days of life, admitted between October 2011 and September 2013 to Emergency Departments of 19 Spanish hospitals, members of the Spanish Paediatric Emergency Research Group of the Spanish Society of Paediatric Emergencies (RISeuP/SPERG). RESULTS: The analysis included 3,401 febrile infants ≤90 days of age with fever without source. There were 896 positive cultures: 766 urine (85.5%), 100 blood (11.2%), 18 cerebrospinal fluid (2%), 10 stool, and 2 umbilical cultures. Among the 3,401 infants included, 784 (23%) were diagnosed with a serious bacterial infection, and 107 of them (3.1%) with an invasive infection. E. coli was the most common pathogen isolated from urine (628; 82%), blood (46; 46%), and cerebrospinal fluid cultures (7; 38.9%), followed by S. agalactiae that was isolated from 24 (24%) blood cultures and 3 (16.7%) cerebrospinal fluid cultures. There were only 2 L. monocytogenes infections. Four children died, and seven had severe complications. CONCLUSIONS: Among infants ≤ 90 days of age with fever without source, E. coli was the most common pathogen isolated from urine, blood, and cerebrospinal fluid cultures.

Rapid Time to Positivity of Cerebrospinal Fluid Culture with Coagulase-Negative Staphylococcus Is More Likely to Reflect a True Infection Than Contamination.

OBJECTIVE: Cerebrospinal fluid (CSF) culture is the gold standard for diagnosing postoperative central nervous system infection. The time to positivity (TTP) of an automated continuous blood culture system may indicate the original concentration of the organism. Coagulase-negative Staphylococcus (CoNS), the common organism recovered in CSF, poses difficulty in differentiating infection from contamination. This study investigated the TTP of CSF culture with CoNS and its relationship to clinical parameters and prognosis. METHODS: Adult neurosurgical patients with CoNS who recovered via the use of CSF culture in BacT/ALERT Pediatric FAN blood culture bottles and were admitted from September 2013 to July 2015 were enrolled. The demographics, clinical and microbiological data, and treatment were reviewed, and the TTP of each culture was retrieved. RESULTS: Thirty-nine adult patients with CoNS recovered from CSF culture were included. The TTP ranged from 7.68 to 57.36 hours. A univariate logistic regression analysis indicated patients with rapid TTP (<21.5 hours) compared with those with longer TTP were more likely to be female, show an effective response to antibiotic therapy within 7 days, have clean-contaminated surgical incisions, and show CSF leak. A multivariate logistic regression analysis indicated that being female, an effective antibiotic therapy within 7 days, and clean-contaminated surgical incisions were independent predictors of rapid TTP. CONCLUSIONS: Targeted antibiotic therapy was more likely to be beneficial to patients with a rapid TTP within 7 days, which suggested that CoNS with a rapid TTP represents the pathogen of central nervous system infection rather than contamination in neurosurgical patients.

Clinical, biological, and microbiological pattern associated with ventriculostomy-related infection: a retrospective longitudinal study.

BACKGROUND: Our aim was to describe the pattern of ventriculostomy-related infection (VRI) development using a dynamic approach. STUDY DESIGN: Retrospective longitudinal study. METHODS: We analyzed the files of 449 neurosurgical patients who underwent placement of external ventricular drain (EVD). During the study period, CSF sampling was performed on a daily base setting. VRI was defined as a positive CSF culture resulting in antibiotic treatment. For VRI patients, we arbitrary defined day 0 (D0) as the day antibiotic treatment was started. In these patients, we compared dynamic changes in clinical and biological parameters at four pre-determined time points: (D-4, D-3, D-2, D-1) with those of D0. For all CSF-positive cultures, we compared CSF biochemical markers' evolution pattern between VRI patients and the others, considered as a control cohort. RESULTS: Thirty-two suffered from VRI. Peripheral white blood cell count did not differ between D-4-D0. Median body temperature, CSF cell count, median Glasgow Coma Scale, CSF protein, and glucose concentrations were significantly different between D-4, D-3, D-2, and D0. At D0, 100 % of CSF samples yielded organisms in culture. The physician caring for the patient decided to treat VRI based upon positive CSF culture in only 28 % (9/32) of cases. In the control cohort, CSF markers' profile trends to normalize, while it worsens in the VRI patients. CONCLUSIONS: We showed that clinical symptoms and biological abnormalities of VRI evolved over time. Our data suggest that VRI decision to treat relies upon a bundle of evidence, including dynamic changes in CSF laboratory exams combined with microbiological analysis.

Types, Risk Factors, Clinical symptoms and Diagnostic Tests of Acute Adult Meningitis in Northern Iran During 2006-2012.

BACKGROUND: Acute bacterial meningitis is a medical emergency condition that requires prompt diagnosis and treatment and otherwise associated with serious morbidity and mortality. AIM: The aim of this study was to assess types, risk factors, clinical symptoms and diagnostic tests of meningitis in hospitalized patients of Mazandaran University of medical sciences hospitals during 2006-2012. MATHERIALS AND METHODS: This is a retrospective descriptive study. Following approval of the ethics committee of Mazandaran University of Medical Sciences, records of adult patients diagnosed with acute meningitis from 2006 to 2012 were extracted from Mazandaran Provincial Health Center and patients attending hospitals affiliated to Mazandaran University of Medical Sciences. STATISTICAL ANALYSIS: Data were analyzed with SPSS-16 using descriptive statistics (frequency, mean, standard deviation, and median). RESULTS: In this study, of the 137 patients with meningitis, 73 (53.9%) were viral, 61 (46%) bacterial, 1 (0.7%) fungal, and 2 (1.4%) unknown. The majority of risk factors in patients were head trauma, upper respiratory infection, and drug addiction. The most common clinical signs were headache, fever, nausea and vomiting, and stiff neck. CONCLUSION: In this study, the incidence of meningitis was much lower than any other country. It could be due to geographic variation or incomplete recording of patient's data. It is recommended to perform a longitudinal study during the coming years on patients with meningitis.

Isolation of bacterial cerebrospinal fluid culture contaminants at a major military medical center.

In recent decades, bacterial meningitis rates have decreased secondary to the success of routine vaccinations. Ironically, the decreased incidence may contribute to the challenge of establishing accurate and timely diagnoses. Studies have suggested that in immunocompetent patients with normal cerebrospinal fluid (CSF) white blood cell counts (WBC), positive CSF cultures may be disregarded as presumed contaminants, making the initial CSF WBC increasingly relevant. This single-institution retrospective study sought to integrate clinical data with positive cultures in an era when CSF contaminants may be more commonly isolated in culture than true pathogens. A total of 7715 adult and pediatric CSF samples from 1995 to 2009 were obtained at a major military medical center. Clinical and laboratory data from 121 positive bacterial cultures were reviewed. Our bacterial CSF contamination rate (false positives) was 0.91% (70/7715). True-positive (TP) CSF cultures totaled 51 (0.66%). Among TPs, 16% (8/51) demonstrated normal CSF cell counts. The notably low 15-year CSF contamination rate of 0.91%, suggests that positive cultures are likely to represent true infection in our institution. We believe efforts to decrease the contamination rate are among the most cost-effective, while targeted clinical re-evaluation for all patients with positive CSF cultures remains vital. In light of this data, a targeted approach to re-evaluating positive cultures while incorporating the clinical context remains prudent.

Human Tuberculous Meningitis Caused by Mycobacterium caprae.

INTRODUCTION: Tuberculous meningitis (TM) causes substantial morbidity and mortality in humans. Human TM has been known to be induced by bacteria from the Mycobacterium tuberculosis complex (MTBC), such as M. tuberculosis and M. bovis. CASE PRESENTATION: We describe a case of meningitis treated with fosfomycin, which showed partial effectiveness in an 80-year-old patient. After a lethal myocardial infarction, M. caprae (MC) was identified in cerebrospinal fluid culture. This isolated acid-fast organism was first identified as MTBC by MTBC-specific PCR (16S rDNA-PCR). Furthermore, species-specific identification of the isolate was done by gyrB PCR-restriction fragment length polymorphism analysis of a part of gyrB DNA. Colony morphology of the isolated MC strain showed dysgonic growth on Lowenstein-Jensen medium. The strain was susceptible to pyrazinamide (PZA). CONCLUSION: This isolated strain was convincingly identified as MC according to the phenotypic and genotypic characteristics and PZA sensitivity. This is the first report of MC causing TM.